Characteristics and complications of anogenital infantile hemangiomas: A multicenter retrospective analysis

Justin D Arnold; SunJung Yoon; Nidhi Shah; Morgan Byrne; Lukas Kieswetter; Cathryn Sibbald; Irene Lara-Corrales; Neha Kinariwalla; Maria C Garzon; Mitchell Braun; Sonal D Shah; Ilona J Frieden; Nicole Travis; Carmen Liy Wong; Alan N Snyder; Lara Wine Lee; Erin K Collier; Marcia Hogeling; Esteban Fernandez Faith; Nicole DeVaul; Anna Yasmine Kirkorian

doi:10.1016/j.jaad.2023.06.049

Characteristics and complications of anogenital infantile hemangiomas: A multicenter retrospective analysis

J Am Acad Dermatol. 2023 Nov;89(5):1015-1021. doi: 10.1016/j.jaad.2023.06.049. Epub 2023 Jul 13.

Authors

Justin D Arnold¹, SunJung Yoon², Nidhi Shah³, Morgan Byrne⁴, Lukas Kieswetter⁵, Cathryn Sibbald⁵, Irene Lara-Corrales⁵, Neha Kinariwalla⁶, Maria C Garzon⁷, Mitchell Braun⁸, Sonal D Shah⁹, Ilona J Frieden⁸, Nicole Travis¹⁰, Carmen Liy Wong¹⁰, Alan N Snyder¹¹, Lara Wine Lee¹¹, Erin K Collier¹², Marcia Hogeling¹², Esteban Fernandez Faith¹³, Nicole DeVaul¹⁴, Anna Yasmine Kirkorian¹⁵

Affiliations

¹ Department of Dermatology, University of California, Irvine, California; Department of Dermatology, George Washington University School of Medicine and Health Sciences, Washington, DC.
² Department of Health Policy and Management, University of Maryland School of Public Health, College Park, Maryland.
³ Department of Dermatology, George Washington University School of Medicine and Health Sciences, Washington, DC.
⁴ Milken Institute School of Public Health, Department of Epidemiology, George Washington University, Washington, DC.
⁵ Division of Dermatology, Department of Paediatrics, The Hospital for Sick Children, Toronto, Ontario, Canada.
⁶ Columbia University, Vagelos College of Physicians and Surgeons, New York, New York.
⁷ Departments of Dermatology and Pediatrics, Columbia University Vagelos College of Physicians & Surgeons, New York, New York.
⁸ Department of Dermatology, University of California San Francisco, San Francisco, California.
⁹ Department of Dermatology, University of California San Francisco, San Francisco, California; Department of Dermatology, Case Western Reserve University School of Medicine, University Hospitals Cleveland Medical Center, Cleveland, Ohio.
¹⁰ Children's Hospital of Eastern Ontario, University of Ottawa, Ottawa, Ontario, Canada.
¹¹ Department of Dermatology and Dermatologic Surgery, Medical University of South Carolina, Charleston, South Carolina.
¹² Department of Medicine, Division of Dermatology, David Geffen School of Medicine, University of California, Los Angeles, California.
¹³ Division of Dermatology, Department of Pediatrics, Nationwide Children's Hospital, The Ohio State University College of Medicine, Columbus, Ohio.
¹⁴ Department of Anatomy and Cell Biology, George Washington University School of Medicine and Health Sciences, Washington, DC.
¹⁵ Department of Dermatology, George Washington University School of Medicine and Health Sciences, Washington, DC; Division of Dermatology, Children's National Hospital, Washington, DC. Electronic address: akirkori@childrensnational.org.

PMID: 37451624
DOI: 10.1016/j.jaad.2023.06.049

Abstract

Background: Infantile hemangiomas (IHs) of the anogenital region remain poorly characterized.

Objective: To examine the distribution, ulceration rate, and associated congenital anomalies of anogenital IHs.

Methods: Retrospective study at 8 tertiary referral centers.

Results: A total of 435 infants with an IH of the anogenital region were enrolled (of which, 319 [73%] were girls). Congenital anomalies were present in 6.4% (n = 28) of infants with an anogenital IH. Segmental or partial segmental anogenital IHs ulcerated in 72% (n = 99 of 138) of infants, whereas 45% (n = 133 of 297) of focal anogenital IHs experienced ulceration (P < .001). In a multivariable logistic regression analysis, segmental or partial segmental morphology (adjusted odds ratio [aOR], 2.70; 95% CI, 1.60-4.64), mixed type (aOR, 3.44; 95% CI, 2.01-6.07), and perianal (aOR, 3.01; 95% CI, 1.53-6.12) and buttocks location (aOR, 2.08; 95% CI, 1.17-3.76) had increased odds of ulceration. Segmental or partial segmental IHs of the genitalia were confined to distinct anatomic territories and were predominantly distributed unilaterally, with a linear demarcation at the perineal raphe.

Limitations: Possible selection bias, given recruitment at tertiary referral centers.

Conclusion: This study improves our understanding of high-risk features of anogenital IHs and demonstrates that genital segmental or partial segmental IHs develop within distinct anatomic territories.

Keywords: LUMBAR syndrome; congenital anomalies; genital development; infantile hemangiomas; ulceration; vascular anomalies.